CASE STUDY Open Access

Cluster headache associated with acute maxillary

sinusitis
Bengt Edvardsson
Abstract
Background: Cluster headache is a primary headache by definition not caused by any known underlying structural
pathology. However, symptomatic cases have been described, for example tumours, particularly pituitary adenomas,
malformations, and infections/inflammations. The evaluation of cluster headache is an issue unresolved.
Case description: I present a case of a 24-year-old patient who presented with a 4-week history of side-locked
attacks of pain located in the left orbit. He satisfied the revised International Classification of Headache Disorders
criteria for cluster headache. His medical and family histories were unremarkable. There was no history of headache.
A diagnosis of cluster headache was made. The patient responded to symptomatic treatment. Low-dose computer
tomography scan after 2 weeks displayed a left-sided acute maxillary sinusitis. The headache attacks resolved
completely after treatment with antibiotics and sinus puncture.
Discussion and evaluation: Although I cannot exclude an unintentional comorbidity, in my opinion, the co-occurrence
of an acute maxillary sinusitis with unilateral headache, in a hitherto headache-free man, points toward the fact that in
this case the cluster headache was caused or triggered by the sinusitis. The headache attacks resolved completely after
the treatment and the patient also remained headache free at the follow-up. The response of the headache to
sumatriptan and other typical cluster headache medications does not exclude a secondary form. Symptomatic cluster
headaches responsive to this therapy have been described. Associated cranial lesions such as infections have been
reported in cluster headache patients and the attacks may be clinically indistinguishable from the primary form.
Conclusions: Neuroimaging, preferably contrast-enhanced magnetic resonance imaging including sinuses should
always be considered in patients with cluster headache despite normal neurological examination. Acute maxillary
sinusitis can present as cluster headache.
Keywords: Cluster headache; Acute maxillary sinusitis; Secondary; Symptomatic; Infection
Background
Cluster headache (CH) is a primary headache, by defin-
ition not caused by any underlying structural pathology
and belonging to the group of trigeminal-autonomic
cephalalgias (Headache Subcommittee of the International
Headache Society 2004). CH is the most frequent syn-
drome in this group. The characteristic symptoms are
strictly unilateral head pain (mainly around orbital and
temporal regions) and associated ipsilateral cranial auto-
nomic features. The headache usually lasts 45 to 90 minutes,
but can range between 15 and 180 minutes. A circannual
and circadian pattern is typical. Symptomatic cases of CH
have been described, for example tumours, particu-
larly pituitary adenomas, malformations, and infections/
inflammations (Cittadini & Matharu 2009). The question
whether patients with CH should undergo neuroimaging
to exclude a causal underlying structural lesion is unre-
solved. I here report a case of acute maxillary sinusitis
the symptoms characteristics of which fully comply with
the criteria of cluster headache (Headache Subcommittee
of the International Headache Society 2004). Symptomatic
CH due to maxillary sinusitis is rare. Previous cases have
been described by Takeshima et al. (Headache 1998;
28:208208) and Molins et al. (Med Clin (Barc) 1989;
92:181183).
Correspondence: Bengt.Edvardsson@med.lu.se
Department of Neurology, Faculty of Medicine, Skane University Hospital,
Lund S-221 85, Sweden
a SpringerOpen Journal
2013 Edvardsson; licensee Springer. This is an open access article distributed under the terms of the Creative Commons
Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction
in any medium, provided the original work is properly cited.
Edvardsson SpringerPlus 2013, 2:509
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Case description
A 24-year-old man presented with a 4-week history of
side-locked attacks of excruciatingly severe stabbing and
boring left-sided pain located in the orbit. The attacks
were associated with nasal obstruction, clear nasal dis-
charge, conjunctival injection and restlessness. No con-
tinuous background pain was identified. The duration of
the attacks was about 40 minutes and the frequency 3 per
24 hours, 5 days a week. There was no history of headache.
His medical and family history was otherwise unremark-
able. He was not on any medications and used no drugs.
Vital signs, physical examination, and neurological examin-
ation were normal. Local tenderness over the sinuses was
not found. Laboratory testing was normal. He satisfied the
revised International Classification of Headache Disorders
criteria for cluster headache. A diagnosis of CH was made
and subcutaneous sumatriptan as well as oxygen 100%
(7 L/min) were prescribed. The patient responded to sub-
cutaneous sumatriptan/oxygen with relief within 20 mi-
nutes. A follow-up was planned. An otologist consultation
was performed to rule out an acute sinusitis. Low-dose
computer tomography scan after 2 weeks displayed a left-
sided acute maxillary sinusitis (Figure 1). A sinus puncture
was performed and it displayed acute inflammation/high
leukocyte count. Bacterial culture displayed Haemophilus
influenzae. The headache attacks resolved completely after
treatment with antibiotics and sinus puncture. A low-dose
computer tomography scan was repeated after 6 weeks and
it displayed normal findings (Figure 2). The prescribed
medication was discontinued. No additional treatment was
given. He remained headache-free and had not experienced
any headache attacks at follow-up after several years.
Discussion
The case study highlights a patient with CH responding
to treatment. Evaluation revealed an acute maxillary si-
nusitis. The patient satisfied the revised International
Classification of Headache Disorders criteria for CH
(Headache Subcommittee of the International Headache
Society 2004). Although I cannot exclude an uninten-
tional comorbidity, in my opinion, the co-occurrence of
an acute maxillary sinusitis with unilateral headache, in
a hitherto headache-free man, points towards the fact
that in this case the CH was caused or triggered by the
sinusitis. The headache attacks resolved completely after
treatment and the patient also remained headache free
at the follow-up after several years. An alternative ex-
planation could be the following: during CH attacks
autonomic symptoms, including nasal congestion, are
commonly observed. Nasal congestion could predispose
the patient to develop an acute sinusitis. A spontaneous
remission of an episodic CH could be misinterpreted as
Figure 1 Low-dose computer tomography scan, showing a left-sided acute maxillary sinusitis.
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being an effect of the antibiotic treatment. However, the
patient remained free of CH attacks at the follow up after
several years and had not previously suffered from CH.
The response of the headache to sumatriptan and other
typical CH medications does not exclude a secondary
form (Ad Hoc Committee on Classification of Headache
1962; Testa et al. 2008). Associated cranial lesions such as
tumours have been reported in CH patients and the at-
tacks may be clinically indistinguishable from the primary
form (Ad Hoc Committee on Classification of Headache
1962; Favier et al. 2007). (Mainardi et al. 2010) identified
156 secondary cluster-like headache cases published from
1975 to 2008. They found in the review that vascular path-
ologies, for example intracranial aneurysms and dural fis-
tulas were the first cause of secondary CH, followed by
tumours and inflammatory/infectious diseases, the latter
accounting for 13.1% of cases. Among the inflammatory/
infectious cases, two cases were associated with sphen-
oidal aspergillosis and one each with ophthalmic herpes
zoster, post infection from herpes simplex and maxillary
sinusitis. The article also reports two cases of cluster-like
headache (not fulfilling the criteria for CH) associated
with sinusitis.
The pathophysiology of CH is not well known. The
most widely accepted theory is that primary CH is char-
acterized by hypothalamic activation with secondary
activation of the trigeminal-autonomic reflex, probably
by a trigeminal-hypothalamic pathway (Cittadini & Matharu
2009). The exact pathophysiology in this case is unknown.
A structural lesion may cause autonomic imbalance, re-
sulting in periodic fluctuations in the activity of the auto-
nomic nervous system, ultimately leading to an attack-wise
presentation of the symptoms (Wilbrink et al. 2009). Dif-
ferences in the individual threshold for triggering the
parasympathetic trigeminal reflexes may also play a role
(Straube et al. 2007). The pain mechanism in secondary
CHs seems ascribable to irritation of pain-sensitive struc-
tures and activation of trigeminal nerve endings (Leone &
Bussone 2009).
Attempts have been made to define red flags indicating
a secondary cause when cluster-like headache appears for
the first time (Mainardi et al. 2010). The authors of that
study emphasize in their report that, at first observation,
50% of patients with secondary CH presented as cases ful-
filling the criteria for CH, perfectly mimicking CH. There-
fore, the likelihood that a secondary cause is responsible
Figure 2 Low-dose computer tomography scan 6 weeks later, showing normal findings.
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for a clinical picture mimicking a primary CH, albeit low,
should always be considered (Mainardi et al. 2010). This
opinion is in accordance with the reviews by (Favier et al.
2007) and by, (Wilbrink et al. 2009) which recommend
neuroimaging in all patients with trigeminal-autonomic
cephalalgias. Magnetic resonance imaging is the preferred
procedure for imaging in CH cases because of its greater
sensitivity to vascular disease, tumour, demyelinating dis-
ease, and infections/inflammations (Wilbrink et al. 2009;
Mainardi et al. 2010).
Conclusions
CH might in rare cases be the presenting symptom of an
acute maxillary sinusitis even in typical forms of that
headache. Neuroimaging, preferably magnetic resonance
imaging including sinuses should always be considered
in patients with CH.
The author takes full responsibility for the data presented
in this study, analysis of the data, conclusions, and conduct
of the research. The author had full access to those data
and has maintained the right to publish any and all data in-
dependent of any third party.
Concerning approval of human studies by the appropri-
ate ethics committee and therefore performed in accord-
ance with the ethical standards laid down in the1964
Declaration of Helsinki: In this case this is not appreciable.
Competing interests
The author declares that there are no competing interests.
Received: 29 April 2013 Accepted: 23 September 2013
Published: 5 October 2013
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doi:10.1186/2193-1801-2-509
Cite this article as: Edvardsson: Cluster headache associated with acute
maxillary sinusitis. SpringerPlus 2013 2:509.
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